AACE Clinical Case Reports最新文献_第4页

Editorial for November/December Issue of AACE Clinical Case Reports AACE临床病例报告十一月/十二月号社论。

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2024-11-01 DOI: 10.1016/j.aace.2024.11.001

Sina Jasim MD, MPH (Editor in Chief)

引用次数: 0

Diabetic Ketoacidosis Without Diabetes Mellitus in Acute Pancreatitis 急性胰腺炎合并糖尿病酮症酸中毒。

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2024-11-01 DOI: 10.1016/j.aace.2024.07.007

Duha Zaffar MD , Aditi Rawat MD , Robert T. Chow MD , Kashif M. Munir MD

Background/Objective

Diabetic ketoacidosis (DKA) is typically but not exclusively seen in patients with a history of diabetes mellitus.

Case Report

This is a case of 39 year-old male who was diagnosed with acute pancreatitis based on characteristic symptoms and positive CT findings on presentation. Laboratory testing revealed elevated serum glucose 251 mg/dL, low serum bicarbonate 8 mmol/L, increased anion gap 21, and elevated serum beta-hydroxybutyrate 9.62 mmol/L. Diagnosis of DKA was made, however patient did not carry a diagnosis of diabetes mellitus. His hemoglobin A1c in hospital was normal at 5.4%. Additionally, follow-up hemoglobin A1c at 4 months and 10 months postdischarge did not imply diabetes mellitus, 5.8% at both time points. The patient who was initially managed with intravenous insulin required no insulin or oral diabetic medication on discharge. All these findings argued against new onset diabetes mellitus.

Discussion

This case explores the potential pathophysiology that underlies this phenomenon including possible transient insulin insufficiency due to beta cell dysfunction from pancreatic inflammation. It also highlights the reversibility and transiency of possible beta cell dysfunction during acute pancreatitis and emphasizes the importance of closely assessing the patients' insulin requirements upon discharge, especially when a prior history of diabetes mellitus is absent.

Conclusion

DKA can occur as a rare complication of acute pancreatitis in a nondiabetic patient. Hyperglycemia associated with acute pancreatitis-induced DKA can be temporary and these patients might not necessarily require insulin upon discharge. Therefore, careful discharge planning is very important in such patients.

背景/目的：糖尿病酮症酸中毒（DKA）通常发生在有糖尿病史的患者身上，但并非唯一：这是一个 39 岁男性的病例，根据其特征性症状和就诊时的 CT 阳性结果，他被诊断为急性胰腺炎。实验室检查显示血清葡萄糖升高 251 mg/dL，血清碳酸氢盐偏低 8 mmol/L，阴离子间隙增加 21，血清 beta-hydroxybutyrate 升高 9.62 mmol/L。诊断结果为 DKA，但患者没有糖尿病诊断。住院期间，他的血红蛋白 A1c 正常，为 5.4%。此外，出院后 4 个月和 10 个月的随访血红蛋白 A1c 均为 5.8%，并不意味着患者患有糖尿病。最初使用静脉注射胰岛素的患者在出院时不需要胰岛素或口服糖尿病药物。所有这些结果都表明，该患者没有新发糖尿病：本病例探讨了这一现象的潜在病理生理学基础，包括胰腺炎症引起的β细胞功能障碍可能导致的一过性胰岛素分泌不足。该病例还强调了急性胰腺炎期间可能出现的β细胞功能障碍的可逆性和短暂性，并强调出院时密切评估患者胰岛素需求的重要性，尤其是在既往无糖尿病史的情况下：结论：DKA 是非糖尿病患者急性胰腺炎的罕见并发症。急性胰腺炎诱发的 DKA 相关高血糖可能是暂时的，这些患者出院时不一定需要胰岛素。因此，为这类患者制定谨慎的出院计划非常重要。

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引用次数: 0

Extensive Deep Vein Thrombosis in a Young Man Taking Tirzepatide for Weight Loss 服用替西帕肽减肥的年轻人广泛深静脉血栓。

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2024-11-01 DOI: 10.1016/j.aace.2024.08.011

Mohammed Fareeduddin Farooqi FRCP , Muhammad Arshad Mehmood FRCP , Maria Khan MBBS , Hafiz Muhammad Salman Pharm D. , Adnan Agha FRCP

Background/Objective

Obesity and rapid weight loss are risk factors for developing deep vein thromboses (DVTs). Our aims were to present a patient who developed extensive DVT after relatively rapid and severe weight loss that followed taking tirzepatide and to raise the awareness among health care professionals regarding the risk of DVT that can be associated with significant weight loss due to these agents.

Case Report

We present the case of a 20-year-old young man, with raised body mass index of >35 kg/m², who was initiated on tirzepatide treatment for weight loss, with 12-kg weight lost over 6 weeks. The patient did not have any risk factors for thrombophilia including family history, any recent travel, immobilization, recent infections, or recent surgeries. He presented with left leg swelling, and physical examination revealed signs of proximal DVT, and ultrasound Doppler and computed tomography venography confirmed extensive left-sided DVT with complete obstruction of the common femoral and iliac veins. He underwent mechanical thrombectomy and was maintained on anticoagulation therapy. His investigations for thrombophilia screening excluded any other cause for DVT, with the etiology attributed to possibly rapid weight loss.

Discussion

Newer and potent glucagon-like peptide 1 receptor agonists like tirzepatide are commonly used nowadays to induce weight loss in obese patients.

Conclusion

Adequate risk assessments and close monitoring should be performed in patients initiating glucagon-like peptide 1 receptor agonists, particularly if they have risk factors for developing venous thromboembolism.

背景/目的：肥胖和体重快速减轻是发生深静脉血栓形成（DVTs）的危险因素。我们的目的是介绍一位在服用替西帕肽后相对快速和严重的体重减轻后发生广泛深静脉血栓的患者，并提高卫生保健专业人员对深静脉血栓风险的认识，这些风险可能与这些药物导致的显著体重减轻有关。病例报告：我们报告了一名20岁的年轻人，体重指数升高，体重指数为bbb35 kg/m2，他开始使用替西帕肽治疗减肥，6周内体重减轻了12 kg。患者没有任何血栓形成的危险因素，包括家族史、近期旅行、固定、近期感染或近期手术。患者表现为左腿肿胀，体格检查显示近端DVT征象，超声多普勒和计算机断层静脉造影证实左侧广泛DVT，股总静脉和髂总静脉完全阻塞。他接受了机械取栓术，并继续接受抗凝治疗。他的血栓筛查调查排除了任何其他导致深静脉血栓形成的原因，病因可能归因于体重迅速减轻。讨论：较新的和有效的胰高血糖素样肽1受体激动剂，如替西肽，目前常用于诱导肥胖患者减肥。结论：对开始使用胰高血糖素样肽1受体激动剂的患者应进行充分的风险评估和密切监测，特别是如果他们有发生静脉血栓栓塞的危险因素。

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引用次数: 0

Medullary Thyroid Carcinoma in the Background of Non-neoplastic Toxic Nodular Goiter 非肿瘤性毒性结节性甲状腺肿背景下的甲状腺髓样癌

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2024-11-01 DOI: 10.1016/j.aace.2024.08.008

Azra Rizwan MBBS, FCPS Medicine, MSc Clinical Research , Malik Saad MBBS, FCPS Medicine, Fellowship in Diabetes and Endocrinology , Saira Fatima MBBS, FCPS Histopathology , Abdullah Ameen MBBS, FCPS Radiology , Shabbir Akhter MBBS, FCPS Surgery

Background/Objective

Medullary thyroid carcinoma (MTC) is an uncommon thyroid cancer (TC), rarely found in hyperfunctioning goiter.

Case Report

We present a case of a woman treated for breast carcinoma (BCA) found to have a benign hyperfunctioning nodular goiter, its likely transformation to MTC, and its treatment. Family history revealed papillary thyroid cancer in her nephew.

Discussion

Most TCs in hyperfunctioning nodules are differentiated carcinomas. Familial MTC or MTC in association with multiple endocrine neoplasia 2 is the expected genetic association in this case.

Conclusion

The association of BCA and MTC may have been coincidental, given the high prevalence of BCA in females. It could have been the result of a common genetic precursor of both tumors and/or treatment modality such as external beam radiation therapy used to treat BCA. This case highlights the importance of considering MTC as a potential diagnosis even in cases of hyperfunctioning nodular goiter. We call for consideration of calcitonin level measurement in the workup of thyroid nodules in select cases. Close follow-up of thyroid nodules, particularly in patients with another primary malignancy, is important because of possible common genotype triggers.

背景/目的：甲状腺髓样癌（MTC）是一种少见的甲状腺癌（TC），在功能性甲状腺肿中很少发现。病例报告：我们提出一个病例的妇女治疗乳腺癌（BCA）发现有良性功能亢进结节性甲状腺肿，其可能转化为MTC，和它的治疗。家族史显示她侄子患有甲状腺乳头状癌。讨论：大多数功能亢进结节的tc为分化癌。家族性MTC或与多发性内分泌瘤2相关的MTC是本病例预期的遗传关联。结论：鉴于BCA在女性中的高患病率，BCA与MTC的关联可能是巧合。它可能是肿瘤和/或治疗方式（如用于治疗BCA的外束放射治疗）的共同遗传前体的结果。本病例强调了考虑MTC作为潜在诊断的重要性，即使在功能亢进的结节性甲状腺肿病例中也是如此。我们呼吁考虑降钙素水平测量在甲状腺结节的检查中选择的情况下。密切随访甲状腺结节，特别是其他原发性恶性肿瘤患者，是很重要的，因为可能有共同的基因型触发。

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引用次数: 0

Successful Radiofrequency Ablation of an Intrathyroidal Parathyroid Adenoma After Failed Parathyroidectomy 甲状旁腺切除术失败后甲状腺内甲状旁腺腺瘤的射频消融成功。

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2024-11-01 DOI: 10.1016/j.aace.2024.08.009

Leslie S. Eldeiry MD , Salomao Faintuch MD , Barry A. Sacks MD

Background/Objective

Radiofrequency ablation (RFA) has been increasingly used as an alternative to surgery in patients with primary hyperparathyroidism who are unable or unwilling to have surgery.

Case Report

We present a case of a 64-year-old woman who had surgery for primary hyperparathyroidism complicated by osteoporosis. Preoperative imaging with 4-dimensional computed tomography scan suggested multigland disease; however, she had persistent hyperparathyroidism after parathyroid exploration. Reevaluation of her imaging studies with ultrasound (US) and US-guided fine needle aspiration for parathyroid hormone washout led to identification of an intrathyroidal parathyroid adenoma that was successfully treated by RFA followed by ethanol injection.

Discussion

The case illustrates the utility of US and US-guided fine needle aspiration for the identification of intrathyroidal parathyroid adenomas, as well as successful treatment of a patient who had failed parathyroid surgery, with a combination of both radiofrequency and percutaneous ethanol ablation, a novel technique.

Conclusion

RFA, with or without ethanol ablation, can be considered as an alternative to surgery for patients with persistent hyperparathyroidism after parathyroidectomy.

背景/目的：射频消融（RFA）越来越多地被用于原发性甲状旁腺功能亢进患者不能或不愿手术的替代手术。病例报告：我们报告了一例64岁的女性，因原发性甲状旁腺功能亢进合并骨质疏松而接受手术。术前4维计算机断层扫描提示多腺体病变；然而，在甲状旁腺探查后，她出现了持续的甲状旁腺功能亢进。通过超声（US）和US引导下细针抽吸检查甲状旁腺激素洗脱，重新评估她的影像学检查，确定了甲状腺内甲状旁腺腺瘤，并通过RFA和乙醇注射成功治疗。讨论：该病例说明了US和US引导的细针穿刺在甲状腺内甲状旁腺腺瘤识别中的应用，以及射频和经皮乙醇消融（一种新技术）联合治疗甲状旁腺手术失败患者的成功。结论：对于甲状旁腺切除术后持续性甲状旁腺功能亢进症患者，RFA可考虑联合或不联合乙醇消融作为手术的替代方案。

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引用次数: 0

Insulin-Induced Severe Lipohypertrophy 胰岛素诱发的严重脂肪肥厚症

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2024-09-01 DOI: 10.1016/j.aace.2024.03.005

引用次数: 0

Noonan Syndrome and Celiac Disease in an Adolescent With Short Stature and Delayed Puberty 一名身材矮小、青春期延迟的青少年患有努南综合征和乳糜泻

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2024-09-01 DOI: 10.1016/j.aace.2024.05.002

Justin Lee BA , Sabitha Sasidharan Pillai MD , Avani Ganta MD , Chanika Phornphutkul MD , Jose Bernardo Quintos MD

Background/Objective

We present an adolescent male with Noonan syndrome (NS) and celiac disease (CD) who attained normal adult height with growth hormone (GH) treatment and gluten-free diet (GFD).

Case Report

A 15 ½ year old healthy male presented with short stature and delayed puberty. His mother and maternal grandmother were short with heights 142.2 cm and 147.3 cm, respectively. Examination showed bilateral epicanthal folds and down slanting eyes like his mother, fifth finger clinodactyly, height 147.5 cm (<1%; standard deviation score, −2.96), growth velocity 2.5 cm/y, weight 48.2 kg (11%; standard deviation score, −1.24), Tanner 2 pubic hair and Tanner 1 genitalia. Midparental target height was 169.1 cm. He had normal screening studies for GH deficiency and thyroid disorders, prepubertal gonadotropins and testosterone levels, and normal total immunoglobulin A, and elevated antitissue transglutaminase immunoglobulin A 134.7units/mL (0-20). Bone age was 13 years. Genetic evaluation revealed heterozygous missense variant of BRAF gene in him and his mother confirming a diagnosis of NS. He was diagnosed with CD by intestinal biopsy. Patient was started on GH therapy and a GFD with subsequent improvement in growth velocit (6.8-12.3 cm/y) and advancement of puberty. The patient stopped GH therapy at 17 ½ years with a height 165.9 cm.

Discussion

Coexistence of NS caused by BRAF missense variant and CD has not been previously reported. Our patient attained normal adult height with GH therapy and GFD.

Conclusion

NS and CD can co-occur and addressing both these disorders can help patients attain normal height potential.

背景/目的我们介绍了一名患有努南综合征（NS）和乳糜泻（CD）的青少年男性患者，通过生长激素（GH）治疗和无麸质饮食（GFD），他的成年身高达到了正常水平。他的母亲和外祖母身高分别为 142.2 厘米和 147.3 厘米。检查结果显示，他和母亲一样有双侧上睑皱褶和下斜眼，五指挛缩，身高 147.5 厘米（<1%；标准偏差分值，-2.96），生长速度 2.5 厘米/年，体重 48.2 千克（11%；标准偏差分值，-1.24），阴毛为 Tanner 2 型，生殖器为 Tanner 1 型。父母的目标身高为 169.1 厘米。他的 GH 缺乏症和甲状腺疾病筛查结果正常，青春期前促性腺激素和睾酮水平正常，总免疫球蛋白 A 正常，抗组织转谷氨酰胺酶免疫球蛋白 A 升高，为 134.7 单位/毫升（0-20）。骨龄为 13 岁。遗传学评估显示，他和他母亲的 BRAF 基因存在杂合错义变异，确诊为 NS。通过肠道活检，他被诊断为 CD。患者开始接受 GH 治疗和 GFD，随后生长速度得到改善（6.8-12.3 厘米/年），青春期提前。患者在 17 岁半时停止了 GH 治疗，身高达到 165.9 厘米。结论NS和CD可同时存在，解决这两种疾病可帮助患者获得正常的身高潜能。

{"title":"Noonan Syndrome and Celiac Disease in an Adolescent With Short Stature and Delayed Puberty","authors":"Justin Lee BA , Sabitha Sasidharan Pillai MD , Avani Ganta MD , Chanika Phornphutkul MD , Jose Bernardo Quintos MD","doi":"10.1016/j.aace.2024.05.002","DOIUrl":"10.1016/j.aace.2024.05.002","url":null,"abstract":"<div><h3>Background/Objective</h3><p>We present an adolescent male with Noonan syndrome (NS) and celiac disease (CD) who attained normal adult height with growth hormone (GH) treatment and gluten-free diet (GFD).</p></div><div><h3>Case Report</h3><p>A 15 ½ year old healthy male presented with short stature and delayed puberty. His mother and maternal grandmother were short with heights 142.2 cm and 147.3 cm, respectively. Examination showed bilateral epicanthal folds and down slanting eyes like his mother, fifth finger clinodactyly, height 147.5 cm (<1%; standard deviation score, −2.96), growth velocity 2.5 cm/y, weight 48.2 kg (11%; standard deviation score, −1.24), Tanner 2 pubic hair and Tanner 1 genitalia. Midparental target height was 169.1 cm. He had normal screening studies for GH deficiency and thyroid disorders, prepubertal gonadotropins and testosterone levels, and normal total immunoglobulin A, and elevated antitissue transglutaminase immunoglobulin A 134.7units/mL (0-20). Bone age was 13 years. Genetic evaluation revealed heterozygous missense variant of <em>BRAF</em> gene in him and his mother confirming a diagnosis of NS. He was diagnosed with CD by intestinal biopsy. Patient was started on GH therapy and a GFD with subsequent improvement in growth velocit (6.8-12.3 cm/y) and advancement of puberty. The patient stopped GH therapy at 17 ½ years with a height 165.9 cm.</p></div><div><h3>Discussion</h3><p>Coexistence of NS caused by <em>BRAF</em> missense variant and CD has not been previously reported. Our patient attained normal adult height with GH therapy and GFD.</p></div><div><h3>Conclusion</h3><p>NS and CD can co-occur and addressing both these disorders can help patients attain normal height potential.</p></div>","PeriodicalId":7051,"journal":{"name":"AACE Clinical Case Reports","volume":"10 5","pages":"Pages 174-178"},"PeriodicalIF":0.0,"publicationDate":"2024-09-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2376060524000476/pdfft?md5=004ad85fce71f9dab13ce526064a2153&pid=1-s2.0-S2376060524000476-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142162751","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

Non–Islet-Cell Tumor Hypoglycemia Secondary to Malignant Phyllodes Tumor of the Breast 继发于乳腺恶性植物瘤的非胰岛细胞肿瘤性低血糖症（NICTH）

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2024-09-01 DOI: 10.1016/j.aace.2024.07.002

Si Min Lee MRCP, MMed, MBBS (Hons) , Timothy Peng Lim Quek MRCP, MMed, MRCPSG, Dip, MSc, DLSHTM, DipRCPath, MBBS (Hons) , Cherng Jye Seow MBBS, DFD, GDFM, MRCP, MRCPS, MCI , Pei Shan Yeo MBBS, MRCP, MMed

Background/Objective

Non–islet cell tumor hypoglycemia (NICTH) is an uncommon condition, of which only a few cases caused by malignant phyllodes tumor of the breast have been reported. We describe a case of NICTH secondary to malignant phyllodes tumor with good response to glucocorticoid therapy.

Case Report

A 62-year-old woman with a rapidly enlarging left breast mass presented with drowsiness and a capillary blood glucose level of 32.4 mg/dL. Her plasma glucose and insulin levels were 36.0 mg/dL (reference range, 72-144 mg/dL) and 0.6 mIU/L (reference range, 0.0-25.0 mIU/L), respectively. Her beta-hydroxybutyrate and c-peptide levels were undetectable. The insulin-like growth factor (IGF)-I and IGF-II levels were 37 μg/L (reference range, 43-220 μg/L) and 1062 ng/mL (reference range, 333-967 ng/mL), respectively, with an IGF-II:IGF-I molar ratio of 29.4. Prednisolone 30 mg per day was initiated with improvement in hypoglycemia. Outpatient flash glucose monitoring profile was stable with mild hypoglycemia (glucose level, 54-68.5 mg/dL) detected 5% of the time. The patient underwent left mastectomy with axillary clearance 4 weeks later. Histology was reported as malignant phyllodes tumor with extensive ductal carcinoma in situ. Prednisolone was stopped after surgery. The patient was treated with letrozole and adjuvant radiotherapy. There was no recurrence of hypoglycemia during the subsequent 24-month follow-up.

Discussion

The mainstay of treatment for NICTH is surgical resection of the culprit tumor. Although glucocorticoid treatment has also been widely used for NICTH, few reports have demonstrated efficacy for NICTH secondary to phyllodes tumor.

Conclusion

We report a rare case of malignant phyllodes tumor of the breast resulting in NICTH and demonstrated good response to glucocorticoids as a bridge to definitive surgery.

背景/目的非胰岛细胞瘤性低血糖症（NICTH）是一种不常见的疾病，其中由乳腺恶性梭形细胞瘤引起的病例仅有少数报道。我们描述了一例继发于恶性梭形细胞瘤的 NICTH 病例，该病例对糖皮质激素治疗反应良好。病例报告：一名 62 岁女性，左侧乳房肿块迅速增大，出现嗜睡和毛细血管血糖水平 32.4 mg/dL。她的血浆葡萄糖和胰岛素水平分别为 36.0 mg/dL（参考范围 72-144 mg/dL）和 0.6 mIU/L（参考范围 0.0-25.0 mIU/L）。她的β-羟丁酸和c-肽水平检测不到。胰岛素样生长因子（IGF）-I和IGF-II水平分别为37微克/升（参考范围：43-220微克/升）和1062纳克/毫升（参考范围：333-967纳克/毫升），IGF-II与IGF-I的摩尔比为29.4。开始使用泼尼松龙，每天 30 毫克，低血糖症状有所改善。门诊闪光灯血糖监测结果稳定，5%的时间检测到轻度低血糖（血糖水平，54-68.5 mg/dL）。4 周后，患者接受了左侧乳房切除术和腋窝清扫术。组织学报告为恶性植物瘤，伴有广泛的导管原位癌。术后停用了泼尼松龙。患者接受了来曲唑治疗和辅助放疗。讨论 NICTH 的主要治疗方法是手术切除原发肿瘤。尽管糖皮质激素治疗也被广泛用于 NICTH，但很少有报道证实其对继发于蝶骨瘤的 NICTH 有疗效。

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引用次数: 0

A Case of Nonislet Cell Tumor Hypoglycemia Due to Metastatic Salivary Myoepithelial Carcinoma 唾液腺肌上皮癌转移导致非小细胞肿瘤性低血糖症一例

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2024-09-01 DOI: 10.1016/j.aace.2024.06.002

Margaret C. Slack MD , Samantha Sovich MD , Chana R. Sachs MD , Dorothy Martinez MD , Run Yu MD

Background/Objective

Nonislet cell tumor hypoglycemia (NICTH) is an uncommon cause of hypoglycemia due to a relative surplus of insulin-like growth factor 2 (IGF-2) or its precursor molecule. The diagnosis is confirmed by an elevated ratio of IGF-2 to insulin-like growth factor 1 (IGF-1). Myoepithelial carcinoma (MECA) is a rare and aggressive salivary gland cancer that has not been previously associated with NICTH.

Case Report

A 63-year-old female with a past medical history of metastatic salivary MECA, type 2 diabetes mellitus previously on metformin, hypertension, and hypothyroidism presented to her oncologist for chemotherapy and was found to have a serum glucose of 30 mg/dL (reference: 65-99). She was admitted for further diagnostic work-up which revealed an insulin level of <1 μU/mL (reference: 3-25), C-peptide <0.5 ng/mL (reference: 1.1-4.3), IGF-1 of 15 ng/mL (reference: 41-279), and IGF-2 of 147 ng/mL (reference: 180-580) with an IGF-2:IGF-1 molar ratio of 10, consistent with NICTH. The patient’s hypoglycemia unfortunately was quite resistant to treatment, requiring a combination of corticosteroids, continuous dextrose infusion, and somatostatin injections. The patient died 3 weeks after presenting with hypoglycemia.

Discussion

Salivary MRCAs commonly contain pleomorphic adenoma gene 1 oncogene rearrangements which are associated with increased IGF-2 production and may predispose patients to hypoglycemia.

Conclusion

This case demonstrates that NICTH can be associated with metastatic salivary MECA. The hypoglycemia in this scenario is challenging to manage and is associated with poor prognosis.

背景/目的非小细胞肿瘤性低血糖症（NICTH）是由于胰岛素样生长因子2（IGF-2）或其前体分子相对过剩而引起的一种不常见的低血糖症。IGF-2与胰岛素样生长因子1（IGF-1）的比值升高即可确诊。病例报告一名 63 岁女性患者，既往病史为转移性唾液腺上皮癌（MECA）、2 型糖尿病，曾服用二甲双胍、高血压和甲状腺功能减退症，因化疗到肿瘤科就诊，发现血糖为 30 mg/dL（参考值：65-99）。她入院接受进一步诊断检查，结果显示胰岛素水平为 1 μU/mL（参考值：3-25），C 肽为 0.5 ng/mL（参考值：1.1-4.3），IGF-1 为 15 ng/mL（参考值：41-279），IGF-2 为 147 ng/mL（参考值：180-580），IGF-2:IGF-1 摩尔比为 10，符合 NICTH。不幸的是，患者的低血糖症非常难治，需要联合使用皮质类固醇、持续输注葡萄糖和注射体生长抑素。讨论唾液腺 MRCA 通常含有多形性腺瘤基因 1 的癌基因重排，这与 IGF-2 生成增加有关，可能导致患者出现低血糖。本病例表明，NICTH 可与转移性唾液腺肿瘤伴发，这种情况下的低血糖症极难处理，且与预后不良有关。

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引用次数: 0

Treatment of Bisphosphonate-Associated Atypical Femur Fracture With a Combination of Teriparatide and a Novel Surgical Technique 特立帕肽与新型外科技术相结合治疗双膦酸盐相关非典型股骨骨折

Q3 Medicine

AACE Clinical Case Reports

Pub Date : 2024-09-01 DOI: 10.1016/j.aace.2024.05.001

Background/Objective

Atypical femur fractures (AFFs) caused by long-term bisphosphonate use are associated with high rates of delayed healing and nonunion.

Case Report

A 64-year-old woman with osteopenia on alendronate for 15 years sustained a displaced left AFF following a fall from standing height. Imaging showed an acute displaced transverse diaphyseal left femur fracture with lateral cortical thickening and beaking. She underwent an open reduction and internal fixation with insertion of a cephalomedullary nail placed in compression mode, utilizing a novel technique involving intraoperative removal of the endosteal hypertrophied cortical bone at the fracture site. Alendronate was stopped and teriparatide was initiated postoperatively. Radiographs at 3.5 months postsurgery showed evidence of normal fracture union with mature callus formation.

Discussion

AFFs caused by prolonged bisphosphonate use have a high rate of delayed healing and nonunion due to abnormal bone remodeling. Use of teriparatide postoperatively has been shown to reduce healing time in small observational studies in surgically treated patients. Our case demonstrates an expedited healing time of 3.5 months using teriparatide combined with a novel surgical technique involving removal of a portion of the abnormally remodeled bone and placement of an intramedullary nail in compression mode.

Conclusion

Our case demonstrates an expedited healing time of 3.5 months compared to the average reported healing time for AFF of 10.7 months, supporting the use of the combination of teriparatide and a novel surgical technique.

背景/目的：长期使用双膦酸盐引起的非典型股骨骨折（AFF）与高延迟愈合率和非愈合率有关。病例报告：一名 64 岁的女性患者患有骨质疏松症，服用阿仑膦酸盐 15 年，一次从站立高度摔倒后，左侧股骨骨折移位。影像学检查显示，左股骨横骺急性移位骨折，外侧皮质增厚并呈蜂窝状。她接受了切开复位和内固定术，在加压模式下植入了头髓内钉，采用了一种新技术，即术中去除骨折部位骨膜内肥厚的皮质骨。术后停用阿仑膦酸钠，并开始使用特立帕肽。术后3.5个月的X光片显示骨折愈合正常，并有成熟的胼胝形成。讨论长期使用双膦酸盐引起的骨折由于骨质重塑异常，延迟愈合和不愈合的发生率很高。小型观察性研究显示，术后使用特立帕肽可缩短手术治疗患者的愈合时间。我们的病例显示，使用特立帕肽并结合一种新的手术技术（切除部分异常重塑的骨骼并在加压模式下放置髓内钉），患者的愈合时间缩短了 3.5 个月。结论我们的病例显示，与所报道的 AFF 平均愈合时间 10.7 个月相比，患者的愈合时间缩短了 3.5 个月，这支持了特立帕肽和新手术技术的结合使用。

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引用次数: 0